Figure 2

Figure 2

katnb1 mutants exhibit cilia defects in foxj1-positive cell lineages

(A–H) SEM imaging of the rhombencephalic ventricle in brains dissected from 3-month-old katnb1^mh102/+ (A; N = 3, n = 9) and katnb1^mh102/mh102 (B; N = 4, n = 11) adults; 6-week-old katnb1^mh102/+ (C; N = 3, n = 9) katnb1^mh102/mh102 (D; N = 3, n = 8) and ptk7a^hsc9/hsc9 (E; N = 3, n = 9) fish; and 3-week-old katnb1^mh102/+ (F; N = 4, n = 14), katnb1^mh102/mh102 (G; N = 4, n = 14) and ptk7a^hsc9/hsc9 (H; N = 3, n = 8) juveniles. Scale bars, 5 μm.

(I and J) Quantification of bulk CSF movement as measured by the distance fluorescent dye travels along the spinal canal (in millimeters; mm), 2 h post-injection into the brain ventricles of experimental fish. (I) katnb1^mh102/+ (A; N = 3, n = 37) and katnb1^mh102/mh102 (A; N = 3, n = 36) zebrafish exhibit no difference in bulk CSF flow rates at 2 weeks of age (p = 0.4828). (J) Significant differences in bulk CSF flow were observed between katnb1^mh102/+ (B; N = 2, n = 19) and katnb1^mh102/mh102 mutant (B; N = 2, n = 12) fish at 3 weeks of age (p = 0.0000006829). Statistical analysis was performed using a two tailed t test.